What can we learn from other illnesses?

[Guest guest]

Journal of Cystic Fibrosis

Volume 2, Issue 1 , March 2003, Pages 58-60

Copyright © 2003 European Cystic Fibrosis Society. Published by Elsevier

Science B. V.

What can we learn from other illnesses?

C. Eiser Cancer Research UK Child and Family Research Group, University

of

Sheffield, Sheffield, UK

I would like to begin by putting forward the idea that there is much to

be gained from a more generic as opposed to a medical model in working

with chronically sick children. I would like to consider the potential

advantages of this generic approach, and then go on to consider more

specifically what can be learned, firstly, from the specific example of

childhood cancer and, secondly, from chronic illness more generally.

Whether we work with children with cancer, cystic fibrosis or any other

chronic condition, we have to be impressed by the child's capacity to

`survive against the odds'. While early work described the problems or

deficits shown by sick children, it is now clear that there is huge

variability in outcomes. Many children show extraordinary resilience and

coping. We are increasingly concerned about quality of life (QOL) as well

as quantity of survival. In expanding on the thesis that much can be

learned from other diseases, I am going to use QOL as an example. Author

1. Introduction––biomedical vs. non-categorical approaches

There is no doubt that there have been impressive advances in medicine

that have resulted in much improved survival for children with many

previously life-threatening conditions. As survival rates have improved,

there has been greater recognition of children's educational, social and

family needs. Traditionally, children with chronic diseases been seen in

terms of specific diagnoses; they are children with cancer, or children

with diabetes. While this is appropriate in some ways, especially with

regard to treatment, it is inadequate in others. Chronic illnesses vary

along a number of dimensions. Some involve demanding self-care rituals,

others very little. Some are life threatening; others more chronic. They

all involve a degree of uncertainty. In a non-categorical framework,

children with different conditions are grouped together, with greater

weight being given to the social and psychological implications of the

condition.For most purposes, it is the consequences of illness that

affect families, not the label. Chronic conditions have consequences for

the child, family, education and health care systems. Often, the label

tells us little about the disease manifestations. One child with

leukaemia may be severely affected by treatment, be unable to return to

school for a year; yet another can sail through treatment in such a way

that parents have difficulty recalling that the child has a

life-threatening condition at all. At the same time, some apparently

different diseases have similar consequences. In both cancer and cystic

fibrosis (cf), children can be small and look physically different from

their peers. For children, the problems associated with physical

appearance can be more important than the labels attached to the

conditions.Consequently, in recent years, there have been calls for more

`non-categorical' or generic approaches to work with these children. The

principal assumptions of the non-categorical approach are that the

consequences of specific disease are independent of the diagnosis. It

follows that if some dimensions of illness cut across different

illnesses, then we can, at least sometimes, benefit from experience

gained in other conditions. In the remainder of this talk, I plan to

focus first on childhood cancer and then more generally on chronic

conditions.

2. The example of childhood cancer

Issues of QOL have been central in assessing the impact of treatment of

children with cancer since the 1960s. At one level, there is not much

point in improving a child's peak flow if it does not make them feel

better. The trouble is that children have unique ways of expressing this

`feel better' factor. The first problem in devising a measure is in the

definition and conceptualisation of the concept. To a large extent,

researchers have drawn on definitions of QOL established in work with

adults.One of the most successful QOL measures has been proposed by Jim

Varni and colleagues [1]. The PedsQL includes five subscales measuring

physical, emotional, social, school and general well-being. As an

example, the physical QOL subscale requires the child or adult to rate

each of eight items on a series of five point Likert scales to indicate

the degree of problem experienced over the last four weeks. These items

range from the very active (running) through to basic aspects of

self-care (Having a bath by him/herself). The measure includes parallel

versions for parents and children, which are normalised, and there are

also developmentally appropriate versions for children of different

ages.In a recent review of the literature, we identified eight measures

specifically for children with cancer. It might seem that this choice is

a good thing, but in practice it has not been helpful. Measures that are

called QOL can include different items and subscale, raising questions

about whether or not they are measuring the same construct. In addition,

there have been objections to using QOL measures on the grounds that they

lack psychometric rigour. Although, there is agreement about the

importance of QOL, there is less agreement about its measurement. This

raises the question that such a poorly defined concept is inadequate.

This conclusion is supported if we look at the status of QOL in work with

children with other chronic conditions.

3. Examples from generally chronic illnesses

Measurement of QOL is not solely the province of those working with

children with cancer. It is generally of interest to all those involved

in work with any chronic condition. So we might next ask what could be

learned from the wider discussion of QOL issues. I am drawing on a review

conducted for the National Health Service in the UK by Morse and

myself [2].In this review, we identified 44 separate measures of QOL.

These included both generic and disease-specific measures, and versions

for parent or child completion separately. Some measures provided

parallel versions for parents and children. We identified 19 generic

measures and 24 disease specific measures. Among these, we were able to

identify just one measure specifically for work with children with cystic

fibrosis.It is possible to learn a great deal from this review. Too

often, we concluded that researchers set about developing a new measure

of QOL, without reviewing the literature generally. Development of new

measures is complex and expensive, and it is silly to fail to learn from

what has been done before. This includes failing to fully appreciate the

views of the child. It is quite inadequate for clinicians and other

health professionals to `brain-storm' the content of measures. Neither is

it appropriate to adapt a measure developed for adults. Adults and

children do not share ideas about the cause, treatment or implications of

illness. Children do not have as sophisticated understanding of language,

or comparable ability to use rating scales. Thus, both the content and

form of measures need to be child-sensitive.

4. Conversely......

So far, I have focused on what can be learned from other illnesses in

terms of the implications for work with children with cystic fibrosis.

However, in planning this talk, it became clear that there was a related

question: what can the rest of the world learn from developments in

cystic fibrosis work? Firstly, both children with cancer and those with

cystic fibrosis need extensive treatments. Decisions about treatment at

home rather than hospital are often justified in terms of cost to the

Health Service, but also it is assumed that families prefer home based

care. Debates about the merits of home or hospital based care are

relevant in the care of children with many chronic conditions, but it is

notably in the literature concerning cf that there have been many formal

attempts at evaluation.As would be expected, there is evidence of

improved clinical outcomes and preferences for home-based care among both

families and clinic staff. Wolter et al. [3] used a QOL measure to assess

adolescents and young adults on three separate occasions during the

course of treatment. At the end of treatment, patients were also asked to

rate the degree of disruption to family, personal life, sleeping and

eating.Both the duration of treatment and clinical outcomes (respiratory

function) were similar between home and hospital-based groups.

Unexpectedly, home based patients reported more fatigue, less feelings of

mastery, and reduced overall QOL compared with those treated in hospital.

However, the home based care group did report improvements in personal,

family, sleeping and eating aspects of QOL. The authors suggested that

increased fatigue was a consequence of trying to keep up with normal life

as well as administer treatments at home.These studies suggest that QOL

considerations can improve our understanding of the merits of home-based

over hospital-based care. It is not that either method of treatment

delivery will have a positive impact on all aspects of QOL, but careful

measurement and research can give insights into how patients manage

different kinds of care. This is potentially useful in explaining details

to new patients and helping them make balanced decisions about, which is

right for them. We can all potentially benefit from this kind of work

being conducted with children with cystic fibrosis.In our enthusiasm to

improve the life of the sick child, we often introduce changes in

treatment delivery without checking whether or not these changes will

result in improved QOL. However, it is impossible to be sure that changes

in treatment delivery have the effects we assume. If treatments are

introduced with the aim to improve QOL, it is essential that this is

tested rather than assumed.

5. Conclusions––what can be learned?

Given my title, I have focused on what can be learned from a generic

approach and taken QOL as an example. But I could have equally focused on

other issues. Parenting sick children is complex. Parents are responsible

for medication and their attitudes to the disease are likely to influence

the child's adjustment and adherence. Parenting is an important clinical

issue that is relevant to all parents regardless of the child's specific

condition. I could also have focused on siblings. Siblings are affected,

but again it probably matters less what the specific disease is, compared

with the general distress and uncertainty common to all conditions. I

could have also focused on school issues. Many children with cancer

experience considerable school absence and compromised learning both as a

consequence of school absence and adverse effects of medical treatment.

Yet, arguments for special educational resources have to be set against

the needs of children with other conditions. It may be much more

profitable to establish educational back up for children with any chronic

disease and this would potentially benefit many more children. There are

limited resources available for the kind of social and educational back

up services that many of us would like to see. The disease-specific model

is essentially counter-productive to the attainment of these resources,

to the extent that they engender competition for resource allocation.

ation may be better than competition.In conclusion, I would like to

thank the organisers of this meeting for inviting me to speak, the

National Health Service HTA programme for funding the review of QOL,

Cancer Research-UK for funding the work with children with cancer, and

the many children and their families who have been willing to take part

in our research.

Becki

YOUR FAVORITE LilGooberGirl

YOUNGLUNG EMAIL SUPPORT LIST

www.topica.com/lists/younglung

Pediatric Interstitial Lung Disease Society

http://groups.yahoo.com/group/InterstitialLung_Kids/

[Guest guest]

Journal of Cystic Fibrosis

Volume 2, Issue 1 , March 2003, Pages 58-60

Copyright © 2003 European Cystic Fibrosis Society. Published by Elsevier

Science B. V.

What can we learn from other illnesses?

C. Eiser Cancer Research UK Child and Family Research Group, University

of

Sheffield, Sheffield, UK

I would like to begin by putting forward the idea that there is much to

be gained from a more generic as opposed to a medical model in working

with chronically sick children. I would like to consider the potential

advantages of this generic approach, and then go on to consider more

specifically what can be learned, firstly, from the specific example of

childhood cancer and, secondly, from chronic illness more generally.

Whether we work with children with cancer, cystic fibrosis or any other

chronic condition, we have to be impressed by the child's capacity to

`survive against the odds'. While early work described the problems or

deficits shown by sick children, it is now clear that there is huge

variability in outcomes. Many children show extraordinary resilience and

coping. We are increasingly concerned about quality of life (QOL) as well

as quantity of survival. In expanding on the thesis that much can be

learned from other diseases, I am going to use QOL as an example. Author

1. Introduction––biomedical vs. non-categorical approaches

There is no doubt that there have been impressive advances in medicine

that have resulted in much improved survival for children with many

previously life-threatening conditions. As survival rates have improved,

there has been greater recognition of children's educational, social and

family needs. Traditionally, children with chronic diseases been seen in

terms of specific diagnoses; they are children with cancer, or children

with diabetes. While this is appropriate in some ways, especially with

regard to treatment, it is inadequate in others. Chronic illnesses vary

along a number of dimensions. Some involve demanding self-care rituals,

others very little. Some are life threatening; others more chronic. They

all involve a degree of uncertainty. In a non-categorical framework,

children with different conditions are grouped together, with greater

weight being given to the social and psychological implications of the

condition.For most purposes, it is the consequences of illness that

affect families, not the label. Chronic conditions have consequences for

the child, family, education and health care systems. Often, the label

tells us little about the disease manifestations. One child with

leukaemia may be severely affected by treatment, be unable to return to

school for a year; yet another can sail through treatment in such a way

that parents have difficulty recalling that the child has a

life-threatening condition at all. At the same time, some apparently

different diseases have similar consequences. In both cancer and cystic

fibrosis (cf), children can be small and look physically different from

their peers. For children, the problems associated with physical

appearance can be more important than the labels attached to the

conditions.Consequently, in recent years, there have been calls for more

`non-categorical' or generic approaches to work with these children. The

principal assumptions of the non-categorical approach are that the

consequences of specific disease are independent of the diagnosis. It

follows that if some dimensions of illness cut across different

illnesses, then we can, at least sometimes, benefit from experience

gained in other conditions. In the remainder of this talk, I plan to

focus first on childhood cancer and then more generally on chronic

conditions.

2. The example of childhood cancer

Issues of QOL have been central in assessing the impact of treatment of

children with cancer since the 1960s. At one level, there is not much

point in improving a child's peak flow if it does not make them feel

better. The trouble is that children have unique ways of expressing this

`feel better' factor. The first problem in devising a measure is in the

definition and conceptualisation of the concept. To a large extent,

researchers have drawn on definitions of QOL established in work with

adults.One of the most successful QOL measures has been proposed by Jim

Varni and colleagues [1]. The PedsQL includes five subscales measuring

physical, emotional, social, school and general well-being. As an

example, the physical QOL subscale requires the child or adult to rate

each of eight items on a series of five point Likert scales to indicate

the degree of problem experienced over the last four weeks. These items

range from the very active (running) through to basic aspects of

self-care (Having a bath by him/herself). The measure includes parallel

versions for parents and children, which are normalised, and there are

also developmentally appropriate versions for children of different

ages.In a recent review of the literature, we identified eight measures

specifically for children with cancer. It might seem that this choice is

a good thing, but in practice it has not been helpful. Measures that are

called QOL can include different items and subscale, raising questions

about whether or not they are measuring the same construct. In addition,

there have been objections to using QOL measures on the grounds that they

lack psychometric rigour. Although, there is agreement about the

importance of QOL, there is less agreement about its measurement. This

raises the question that such a poorly defined concept is inadequate.

This conclusion is supported if we look at the status of QOL in work with

children with other chronic conditions.

3. Examples from generally chronic illnesses

Measurement of QOL is not solely the province of those working with

children with cancer. It is generally of interest to all those involved

in work with any chronic condition. So we might next ask what could be

learned from the wider discussion of QOL issues. I am drawing on a review

conducted for the National Health Service in the UK by Morse and

myself [2].In this review, we identified 44 separate measures of QOL.

These included both generic and disease-specific measures, and versions

for parent or child completion separately. Some measures provided

parallel versions for parents and children. We identified 19 generic

measures and 24 disease specific measures. Among these, we were able to

identify just one measure specifically for work with children with cystic

fibrosis.It is possible to learn a great deal from this review. Too

often, we concluded that researchers set about developing a new measure

of QOL, without reviewing the literature generally. Development of new

measures is complex and expensive, and it is silly to fail to learn from

what has been done before. This includes failing to fully appreciate the

views of the child. It is quite inadequate for clinicians and other

health professionals to `brain-storm' the content of measures. Neither is

it appropriate to adapt a measure developed for adults. Adults and

children do not share ideas about the cause, treatment or implications of

illness. Children do not have as sophisticated understanding of language,

or comparable ability to use rating scales. Thus, both the content and

form of measures need to be child-sensitive.

4. Conversely......

So far, I have focused on what can be learned from other illnesses in

terms of the implications for work with children with cystic fibrosis.

However, in planning this talk, it became clear that there was a related

question: what can the rest of the world learn from developments in

cystic fibrosis work? Firstly, both children with cancer and those with

cystic fibrosis need extensive treatments. Decisions about treatment at

home rather than hospital are often justified in terms of cost to the

Health Service, but also it is assumed that families prefer home based

care. Debates about the merits of home or hospital based care are

relevant in the care of children with many chronic conditions, but it is

notably in the literature concerning cf that there have been many formal

attempts at evaluation.As would be expected, there is evidence of

improved clinical outcomes and preferences for home-based care among both

families and clinic staff. Wolter et al. [3] used a QOL measure to assess

adolescents and young adults on three separate occasions during the

course of treatment. At the end of treatment, patients were also asked to

rate the degree of disruption to family, personal life, sleeping and

eating.Both the duration of treatment and clinical outcomes (respiratory

function) were similar between home and hospital-based groups.

Unexpectedly, home based patients reported more fatigue, less feelings of

mastery, and reduced overall QOL compared with those treated in hospital.

However, the home based care group did report improvements in personal,

family, sleeping and eating aspects of QOL. The authors suggested that

increased fatigue was a consequence of trying to keep up with normal life

as well as administer treatments at home.These studies suggest that QOL

considerations can improve our understanding of the merits of home-based

over hospital-based care. It is not that either method of treatment

delivery will have a positive impact on all aspects of QOL, but careful

measurement and research can give insights into how patients manage

different kinds of care. This is potentially useful in explaining details

to new patients and helping them make balanced decisions about, which is

right for them. We can all potentially benefit from this kind of work

being conducted with children with cystic fibrosis.In our enthusiasm to

improve the life of the sick child, we often introduce changes in

treatment delivery without checking whether or not these changes will

result in improved QOL. However, it is impossible to be sure that changes

in treatment delivery have the effects we assume. If treatments are

introduced with the aim to improve QOL, it is essential that this is

tested rather than assumed.

5. Conclusions––what can be learned?

Given my title, I have focused on what can be learned from a generic

approach and taken QOL as an example. But I could have equally focused on

other issues. Parenting sick children is complex. Parents are responsible

for medication and their attitudes to the disease are likely to influence

the child's adjustment and adherence. Parenting is an important clinical

issue that is relevant to all parents regardless of the child's specific

condition. I could also have focused on siblings. Siblings are affected,

but again it probably matters less what the specific disease is, compared

with the general distress and uncertainty common to all conditions. I

could have also focused on school issues. Many children with cancer

experience considerable school absence and compromised learning both as a

consequence of school absence and adverse effects of medical treatment.

Yet, arguments for special educational resources have to be set against

the needs of children with other conditions. It may be much more

profitable to establish educational back up for children with any chronic

disease and this would potentially benefit many more children. There are

limited resources available for the kind of social and educational back

up services that many of us would like to see. The disease-specific model

is essentially counter-productive to the attainment of these resources,

to the extent that they engender competition for resource allocation.

ation may be better than competition.In conclusion, I would like to

thank the organisers of this meeting for inviting me to speak, the

National Health Service HTA programme for funding the review of QOL,

Cancer Research-UK for funding the work with children with cancer, and

the many children and their families who have been willing to take part

in our research.

Becki

YOUR FAVORITE LilGooberGirl

YOUNGLUNG EMAIL SUPPORT LIST

www.topica.com/lists/younglung

Pediatric Interstitial Lung Disease Society

http://groups.yahoo.com/group/InterstitialLung_Kids/